Genetic Information

Gene & Transcript Details

Gene
RUNX1
Transcript
NM_001754.4 MANE Select
Total Exons
Reference Sequence
NC_000021.8
Alternative Transcripts
IDStatusDetails
NM_001754.3 Alternative 6190 nt | 400–1842
NM_001754.4 RefSeq Select 5967 nt | 191–1633
NM_001754.5 MANE Select 5971 nt | 195–1637

Variant Details

HGVS Notation
NM_001754.4:c.1237dup
Protein Change
S413Ffs*187
Location
Exon 9 (Exon 9 of )
9
5'Exon Structure3'
Functional Consequence
Loss of Function
Alternate Identifiers

Clinical & Population Data

Population Frequency

gnomAD
Global Frequency
0.0 in 100,000
Extremely Rare
ACMG Criteria Applied PM2
This variant is absent or extremely rare in population databases (PM2 criteria applies).

ClinVar

Open
Classification
Unknown
0 publications
Clinical Statement

""

COSMIC Somatic Evidence

Open
COSMIC ID
Recurrence
0 occurrences
PM1 Criteria
Not Applied
COSMIC Database Preview
COSMIC Preview
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Functional Impact & Domains

Functional Domain

Hotspot Status
Not a hotspot
Domain Summary
This variant is not located in a mutational hotspot or critical domain.
Related Variants in This Domain
No evidence of other pathogenic variants at this position in gene RUNX1.

Functional Studies & Therapeutic Relevance

Functional Summary

The RUNX1 S413Ffs*187 variant is a truncating mutation in the RUNX1 gene, a tumor suppressor involved in hematopoietic differentiation. Functional evidence indicates that truncating mutations in RUNX1 can inhibit its function, contributing to oncogenesis and predisposing individuals to hematologic malignancies such as leukemia, lymphomas, and myelodysplastic syndromes.

Database Previews
OncoKB
OncoKB Preview

Click on previews to view full database entries. External databases may require institutional access.

Computational Analysis

Pathogenicity Predictions

SpliceAI
Predictor Consensus
Unknown
PP3 Applied
No
REVEL Score
0.0
Threshold: ≥0.75 = PP3 applied

SpliceAI Scores

Window: ±500bp
Effect Type Score Position
- Acceptor Loss (AL) 0.0 270 bp
- Donor Loss (DL) 0.0 -357 bp
+ Acceptor Gain (AG) 0.0 186 bp
+ Donor Gain (DG) 0.0 456 bp
High impact (≥0.5) Medium impact (0.2-0.49) Low impact (<0.2)

VCEP Guidelines

Applied ACMG/AMP Criteria (VCEP Specific)

Filter Criteria:
PVS1

PVS1 (Very Strong)

According to VCEP guidelines, the rule for PVS1 is: "Very Strong Strength: Very Strong Per modified RUNX1 PVS1 decision tree for SNVs and CNVs and table of splicing effects." The evidence for this variant shows: NM_001754.4:c.1237dupT causes a frameshift leading to a premature termination codon and is not located in the last exon. Therefore, this criterion is applied at Very Strong strength because it is a null variant (frameshift) in RUNX1, where loss of function is a known mechanism of disease.

PS1

PS1 (Not Applied)

According to VCEP guidelines, the rule for PS1 is: "Strong Strength: Same amino acid change as a previously established pathogenic variant regardless of nucleotide change." The evidence for this variant shows: S413Ffs*187 does not match any previously established pathogenic amino acid change. Therefore, this criterion is not applied.

PS2

PS2 (Not Applied)

According to VCEP guidelines, the rule for PS2 applies to de novo occurrences (Moderate = ≥1 proven; Supporting = ≥1 assumed). The evidence for this variant shows: No data on de novo occurrence. Therefore, this criterion is not applied.

PS3

PS3 (Not Applied)

According to VCEP guidelines, the rule for PS3 states: "Not applicable if variant meets PVS1." The evidence for this variant shows: PVS1 has been applied at Very Strong. Therefore, PS3 is not applied.

PS4

PS4 (Not Applied)

According to VCEP guidelines, the rule for PS4 is: Strong = ≥4 probands, Moderate = 2–3 probands, Supporting = 1 proband. The evidence for this variant shows: No proband data reported. Therefore, this criterion is not applied.

PM1

PM1 (Not Applied)

According to VCEP guidelines, the rule for PM1 is: "Moderate Strength: Variant affecting one of the specified RHD residues; Supporting Strength: Variant affecting other residues 89–204." The evidence for this variant shows: S413Ffs*187 lies outside the RHD (residues 49–187). Therefore, this criterion is not applied.

PM2

PM2 (Supporting)

According to VCEP guidelines, the rule for PM2 is: "Supporting Strength: Variant must be completely absent from all population databases." The evidence for this variant shows: Not found in gnomAD or other population databases (MAF=0%). Therefore, this criterion is applied at Supporting strength because the variant is absent from controls.

PM3

PM3 (Not Applied)

According to standard ACMG guidelines, the rule for PM3 applies to recessive disorders requiring observation in trans with another pathogenic variant. The evidence for this variant shows: No evidence of biallelic inheritance in a recessive context. Therefore, this criterion is not applied.

PM4

PM4 (Not Applied)

According to standard ACMG guidelines, the rule for PM4 applies to protein length changes due to in-frame insertions or deletions. The evidence for this variant shows: It is a frameshift duplication, not an in-frame indel. Therefore, this criterion is not applied.

PM5

PM5 (Supporting)

According to VCEP guidelines, the rule for PM5 is: "PM5_supporting is also applied to nonsense/frameshift variants that are downstream of c.98." The evidence for this variant shows: c.1237dupT results in a frameshift S413Ffs*187 downstream of codon 98. Therefore, this criterion is applied at Supporting strength because it is a truncating variant downstream of c.98.

PM6

PM6 (Not Applied)

According to VCEP guidelines, the rule for PM6 applies to assumed de novo occurrences. The evidence for this variant shows: No data on de novo occurrence. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to VCEP guidelines, the rule for PP1 applies to segregation in multiple affected family members. The evidence for this variant shows: No segregation data. Therefore, this criterion is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG guidelines, the rule for PP2 applies to missense variants in a gene with low benign missense rate. The evidence for this variant shows: It is not a missense variant. Therefore, this criterion is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines, the rule for PP3 applies to missense or intronic variants with computational predictions. The evidence for this variant shows: It is a frameshift and SpliceAI score 0, which does not predict a splice impact. Therefore, this criterion is not applied.

PP4

PP4 (Not Applied)

According to standard ACMG guidelines, the rule for PP4 applies when a highly specific phenotype is observed. The evidence for this variant shows: No phenotype data provided. Therefore, this criterion is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, the rule for PP5 applies to variant classified as pathogenic by a reputable source without primary data. The evidence for this variant shows: Not found in ClinVar or other expert reports. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines, the rule for BA1 applies to variants with MAF≥0.15%. The evidence for this variant shows: MAF=0%. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines, the rule for BS1 applies to MAF between 0.015% and 0.15%. The evidence for this variant shows: MAF=0%. Therefore, this criterion is not applied.

BS2

BS2 (Not Applied)

According to standard ACMG guidelines, the rule for BS2 applies when variant is observed in healthy individuals. The evidence for this variant shows: No such data. Therefore, this criterion is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines, the rule for BS3 applies to normal functional assay results. The evidence for this variant shows: Functional studies indicate a damaging effect. Therefore, this criterion is not applied.

BS4

BS4 (Not Applied)

According to standard ACMG guidelines, the rule for BS4 applies to lack of segregation in multiple affected families. The evidence for this variant shows: No segregation data. Therefore, this criterion is not applied.

BP1

BP1 (Not Applied)

According to standard ACMG guidelines, the rule for BP1 applies to missense variants in a gene where only truncating variants cause disease. The evidence for this variant shows: It is a truncating variant. Therefore, this criterion is not applied.

BP2

BP2 (Not Applied)

According to standard ACMG guidelines, the rule for BP2 applies when variant is observed in trans with a pathogenic variant for a dominant gene. The evidence for this variant shows: No such data. Therefore, this criterion is not applied.

BP3

BP3 (Not Applied)

According to standard ACMG guidelines, the rule for BP3 applies to in‐frame indels in repetitive regions. The evidence for this variant shows: It is a frameshift duplication, not a repetitive-region indel. Therefore, this criterion is not applied.

BP4

BP4 (Not Applied)

According to VCEP guidelines, the rule for BP4 applies to benign computational evidence (REVEL<0.50 or SpliceAI≤0.20). The evidence for this variant shows: Although SpliceAI=0, the variant causes a frameshift leading to loss of function. Therefore, this criterion is not applied.

BP5

BP5 (Not Applied)

According to standard ACMG guidelines, the rule for BP5 applies when variant is found in a patient with an alternate molecular cause. The evidence for this variant shows: No alternate cause reported. Therefore, this criterion is not applied.

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, the rule for BP6 applies to non‐publication of primary data by a reputable source. The evidence for this variant shows: No such reports. Therefore, this criterion is not applied.

BP7

BP7 (Not Applied)

According to VCEP guidelines, the rule for BP7 applies to synonymous or intronic variants with SpliceAI≤0.20. The evidence for this variant shows: It is a frameshift variant. Therefore, this criterion is not applied.